Wessels Marja W, Catsman-Berrevoets Coriene E, Mancini Grazia M S, Breuning Martijn H, Hoogeboom Jeanette J M, Stroink Hans, Frohn-Mulder Ingrid, Coucke Paul J, Paepe Anne De, Niermeijer Martinus F, Willems Patrick J
Department of Clinical Genetics, Erasmus University Medical Centre, Rotterdam, The Netherlands.
Am J Med Genet A. 2004 Dec 1;131(2):134-43. doi: 10.1002/ajmg.a.30272.
Arterial tortuosity syndrome (ATS) is a rare condition with autosomal recessive inheritance characterized by connective tissue abnormalities. The most specific clinical findings are cardiovascular anomalies including tortuosity, lengthening, aneurysm, and stenosis formation of major arteries. Also ventricular hypertrophy is frequently present. Other anomalies are skin hyperextensibility and cutis laxa, joint laxity or contractures of the joints, and inguinal herniae. Histology shows disruption of elastic fibers of the media. These features suggest that ATS is a connective tissue disorder. A biochemical or molecular defect has not yet been identified. We describe here nine additional ATS patients from three consanguineous Moroccan families and review a total of 35 patients with this uncommon condition.
动脉迂曲综合征(ATS)是一种罕见的常染色体隐性遗传病,其特征为结缔组织异常。最具特异性的临床发现是心血管异常,包括大动脉的迂曲、延长、动脉瘤和狭窄形成。此外,心室肥厚也很常见。其他异常包括皮肤过度伸展和皮肤松弛、关节松弛或关节挛缩以及腹股沟疝。组织学显示中膜弹性纤维断裂。这些特征表明ATS是一种结缔组织疾病。尚未确定生化或分子缺陷。我们在此描述来自三个摩洛哥近亲家庭的另外9例ATS患者,并对总共35例患有这种罕见疾病的患者进行综述。